Ö. Yılmaz et al. Primary ovarian ectopic pregnancy 121
Dicle Tıp Derg / Dicle Med J www.diclemedj.org Cilt / Vol 40, No 1, 121-123
1 Department of Obstetrics and Gynecology, Şişli Etfal Training and Research Hospital, Istanbul, Turkey
2 Department of Pathology, Şişli Etfal Training and Research Hospital, Istanbul, Turkey Yazışma Adresi /Correspondence: Suna Kabil Kucur,
Dept. Obstetrics and Gynecology, Şişli Etfal Training and Research Hospital, Istanbul, Turkey Email: dr.suna@hotmail.com Geliş Tarihi / Received: 17.08.2012, Kabul Tarihi / Accepted: 06.10.2012
Copyright © Dicle Tıp Dergisi 2013, Her hakkı saklıdır / All rights reserved
Dicle Tıp Dergisi / 2013; 40 (1): 121-123
Dicle Medical Journal doi: 10.5798/diclemedj.0921.2013.01.0237
CASE REPORT / OLGU SUNUMU
Diagnosis and clinical approach in primary ovarian ectopic pregnancy: A case report and review of the literature
Primer ovaryan ektopik gebelikte tanı ve klinik yaklaşımı: Bir olgu sunumu ve literatürün gözden geçirilmesi
Özge Yılmaz1, Suna Kabil Kucur1, Duygu Yardım1, İnci Davas1, Nedim Polat2
ÖZET
Primer ovaryan ektopik gebelik oldukça nadir görülüp tüm dış gebeliklerin %0,5-1’ini oluşturur. İnsidansı 1/7000- 40000 gebeliktir. Spontan bir siklusta hiçbir predispozan faktör olmaksızın 40 yaşında bir hastada oluşmuş bir primer ovaryan ektopik gebelik olgusu sunacağız. Hasta akut batın belirti ve bulgularıyla preşok durumda acil kli- niğimize başvurdu. Transvajinal ultrasonografide 30 mm çapında kistik sol ovaryan kitle tespit edildi. Müracaatın- da hemoglobin seviyesi 8,4g/dl ve serum bHCG seviyesi 2548 mlU/ml idi. Acil laparotomi ve over kama rezeksi- yonu uygulandı. Histopatolojik olarak alınan materyalin yapılan çoklu kesilerinde birçok alanda over dokusu içine invaze olmuş koryon villusları izlenmiştir. Bu nadir olgu- nun etyolojisi, ayırıcı tanısı, klinik tanısı hakkında bilgi ve- rilerek literatür gözden geçirilmiştir.
Anahtar kelimeler: Ektopik, gebelik, over ABSTRACT
Primary ovarian pregnancy is a rare type of extrauterine pregnancy with an incidence of 0.5-1% of all ectopic preg- nancies. The incidence ranges from 1/40000 to 1/7000 deliveries. We report a primary ovarian pregnancy in a spontaneus conception cycle in a 40-year-old woman with no predisposing factor. The patient was admitted to the emergency clinic with signs and symptoms of acute abdomen with heamoperitoneum , in a preshock state. An emergency laparotomy and wedge resection of the ovary was performed. Histopathologic examination showed chorion villi embedded in the ovarian tissue in multiple sections in different areas of the material. This is a discus- sion about this rare phenomenon, its ethiology, differential and clinical diagnosis and a review of literature.
Key words: Ectopic, pregnancy, ovary
INTRODUCTION
Primary ovarian pregnancy is one of the rarest types of ectopic pregnancy where the gestational sac is im- planted within the ovary. It represents about 0.5-1%
of all ectopic pregnancies and the incidence ranges from 1/40000 to 1/7000 deliveries. It is responsible for 10% of pregnancy related deaths. The diagnos- tic criteria were described in 1878 by Spiegelberg.1 Unlike the tubal gestation ovarian pregnancy is as- sociated with neither PID nor infertility. The only risk factor associated with the development of an ovarian pregnancy is the current use of IUD.2,3 Mis- diagnosis is common; because it is confused with a ruptured corpus luteum in 75% cases. Usually first clinical presentation is shock with hemoperitoneum
requiring emergency surgery. In recent years, the incidence is increased because of the assisted repro- ductive techniques and wider use of IUD. We re- port a primary ovarian pregnancy in a spontaneous conception cycle in a 40 year old woman with no predisposing factor.
CASE REPORT
A 40-year-old, gravida 5 para 5 woman was admit- ted to the emergency service with a sudden onset of sharp low abdominal pain and faintness. She had no history of pelvic inflammatory disease, abor- tions, and use of intrauterine devices. She was pale.
She had rebound tenderness on the left abdominal
Ö. Yılmaz et al. Primary ovarian ectopic pregnancy 122
Dicle Tıp Derg / Dicle Med J www.diclemedj.org Cilt / Vol 40, No 1, 121-123 region. On bimanual vaginal examination the uter-
us was enlarged to 7-week pregnancy in size and a tender mass was palpated above the left fornix.
On transvaginal ultrasonographic examination, the uterine cavity was empty, endometrium thickness was 13.1 mm and the right ovary was normal. In the left ovary, there was a ovarian cyst sized 25x30 mm with coagulum like sonographic appearance visual- ized around it and there seemed to be some amount of free fluid in the Douglas pouch. The blood pres- sure of the patient was 80/50 temperature was 37.0°C and pulse rate was 118/min being fillform in nature. In the complete blood count hemoglobin:
10.2 g/dL, hematocrit:33.1%, βhCG level was 2548 mlU/mL. With these clinical and laboratory findings the patient was prediagnosed as ectopic pregnancy and an emergency laparotomy was performed. On the exploration of the abdomen with a Pfannenstiel incision, the uterus was normal with no evidence of congenital anomaly, endometriosis or chronic in- flammation. The right ovary and the right fallopian tube were also normal. The left fallopian tube with its fimbrial end was intact and normal; however, there was about 200 mL clotted blood in the abdom- inal cavity. The left ovary was bleedy and cystic in appearance and some amount of placental material was observed when the coagulum on the surface of the ovary was removed. There was no adhesion or pathology in the pelvic cavity. Wedge resection was performed to the left ovary. After the peritoneal cleaning, abdomen was closed. The postoperative hemoglobin was 8.4 g/dL, hematocrit was 27.2%.
Figure 1. Chorion villi seen in the ulcerated kongested ovarian stroma H&E X40
The patient was discharged from the hospital on the third postoperative day without any complication.
The βhCG level was 18.6 mIU/mL on the postop- erative tenth day. Histopathological examination showed a primary ovarian ectopic pregnancy with chorion villi embedded in the ulcerated congested ovarian stroma (Figure 1).
DISCUSSION
Primary ovarian pregnancy is a rare phenomenon and its clinical diagnosis is very difficult. It de- serves an additional afford not to be misdiagnosed as it leads to hemodynamic instability much more than the tubal pregnancies. Most cases represent as ovarian cysts.4 Invasion of the trophoblastic cells of the ovarian tissue on the 6th day is followed by the invasion of the ovarian artery. Although ovarian pregnancies rupture by the 40th gestational day, re- ports of those progressing into the 3rd trimester even to live births have been established. Seki et al.5 de- scribed an ovarian-pregnancy case of 30 weeks of gestation diagnosed at cesarean section. Shahabud- din and Chowdhury6 reported a case of heterotopic pregnancy, the ectopic focus located in the ovary proceeding up to term. There was another case of an ovarian pregnancy by Belfar et al.7 reporting a sur- vived neonate until term. Studzinski et al.8 reported an advanced ovarian pregnancy case with the reten- tion of a dead fetus for more than a year. The risk factors of the tubal pregnancy do not correspond with the risk factors of the ovarian pregnancy. The only risk factor associated with the development of the ovarian pregnancy is the current IUD use. Raziel et al.9 reported 20 series of ovarian pregnancy cases 18 of which had the history of IUD use. However in a series of 54 ovarian pregnancy study reported by Shiau et al.10 only 13% of patients were current IUD users. In that study, they concluded that neither IUD use nor PID seemed to have a role in the oc- currence of the reviewed ovarian pregnancies. Cri- teria for ovarian pregnancy diagnosis is described in 1878 by Spiegelberg (i) the fallopian tube on the effected side must be intact; (ii) the gestational sac must occupy the normal position of the ovary; (iii) the ovary must be connected to the uterus by the ovarian ligament; and (iv) ovarian tissue must be lo- cated in the sac wall.1 All the criteria above are ful- filled in the case we presented. Ovarian pregnancies are classified as primary or secondary depending on
Ö. Yılmaz et al. Primary ovarian ectopic pregnancy 123
Dicle Tıp Derg / Dicle Med J www.diclemedj.org Cilt / Vol 40, No 1, 121-123 the implantation site of the conception material,11 If
the ovum is fertilized within the follicle it is called primary ovarian pregnancy; however, if the fertil- ization takes place in the fallopian tube and second- arily implants on the ovarian tissue it is accepted as secondary ovarian pregnancy. Primary ovarian pregnancy is considered to be due to an ovulatory dysfunction. William and Norris12-13 stated that the tube must be intact and has no evidence of gesta- tion and the ovarian tissue must be observed around the gestational sac at several positions to distinguish the primary from secondary ovarian pregnancy. The ovarian pregnancy is definitely diagnosed perop- eratively. Misdiagnosis of this entity is common because it is confused with a ruptured corpus lu- teum in up to 75% of cases. It is debated that the frequency of ovarian pregnancy is underestimated since the medical therapy with methotrexate is used for suspected tubal pregnancies. This underestima- tion is balanced by more suspicion of the possibility of an ovarian pregnancy. Improvements in ultra- sonography and use of more sensitive radioimmu- noassay for βhCG detection lead to more accurate diagnosis of ovarian pregnancy cases. Although in the past culdocentesis was a very valuable tool for the diagnosis, hemoperitoneum can be visualized by vaginal ultrasonography. Cacciatore et al.14 re- ported that with combined use of βhCG test and ul- trasound the ectopic pregnancies can be diagnosed with 96% sensitivity and 100% specificity. Patients still present with circulatory collapse despite mod- ern diagnostic modalities. The traditional method for management of an ovarian pregnancy is wedge resection or the removal of the effected adnexa by laparoscopy or laparotomy.15 Systemic methotrex- ate admiration has become apparent in the recent years. But, it is not the first-line treatment of choice for ovarian pregnancy even if the patient is candi- date meeting the criteria for medical treatment. In our case the patient was hemodinamically unstable so, an emergency laparotomy and wedge resection of the effected ovary was performed.
In conclusion the primary ovarian pregnancy is a rare entity. There are a few reports about a series of cases of ovarian pregnancies, the largest one in- cluding 54 cases. There might not be predisposing factors as in our case. The diagnosis is now easier because of the improved diagnostic modalities. Ul-
trasonographic appearance of an ovarian cyst in a patient with suspected ectopic pregnancy should im- ply us an ovarian pregnancy. The treatment is being more conservative due to earlier diagnosis. Despite these, many patients are admitted to the hospitals under hemodynamically unstable conditions when the conservative treatment is usually not possible.
With the help of ultrasounds having better imaging quality it is now becoming more probable to make the diagnosis preoperatively.
REFERENCES
1. Spiegelberg O. Zur Casuistik der Ovarialschwangerschaft.
Arch Gynaekol 1878;13:73-6
2. Berek & Novak’s Gynecology, fourteenth edition, 2007:627- 8.
3. Raziel A, Schachter M, Mordechai E, Friedler S, Panski M, Ron-El R. Ovarian pregnancy a 12-year experience of 19 cases in one institution. Eur J Obstet & Gynecol Reprod Biology 2004;114:92-6.
4. Kilic N, Demir B, Arslan A, Ozaydin M. Ovarian Pregnancy:
Case report. Dicle Medical Journal 2002;29: 85-9.
5. Seki H, Kuromaki K, Takeda S, Kinoshita K. Ovarian preg- nancy diagnosed in the third trimester: a case report. J Ob- stet Gynecol Res 1997;23:543-6.
6. Shahabuddin AK, Chowdhury S. Primary term ovarian preg- nancy superimposed by intrauterine pregnancy: a case re- port. J Obstet Gynecol Res 1998;24:109-14.
7. Belfar H, Heller K, Edelstone DI, et al. Ovarian pregnancy resulting in a surviving neonate.Ultrasound findings. J Ul- trasound Med 1991;10:465-7.
8. Studzinski Z, Branika D, Filipezak A, et al. Prolonged ovar- ian pregnancy: a case report. Ginekol Pol 1999;70:33-5.
9. Raziel A, Golan A, Pansky M, et al. Ovarian pregnancy-a report of 20 cases in one institution. Am Obstet Gynecol 1990;163:1182-5.
10. Shiau CS, Hsieh CL, Chang MY. Primary ovarian pregnan- cy. Int J Gynecol Obstet 2007;96:127-30.
11. Check JH, Chase JS. Ovarian pregnancy with contralateral corpus luteum. Am J Obstet Gynecol 1986;154:155-6.
12. Williams JV. Obstetrics, Ovarian Pregnancy. Appleton-Cen- tury-Crott, New York. 1903:573.
13. Norris CC. Primary ovarian pregnancy and the report of a case combined with intrauterine pregnancy. Surg Gynecol Obstet 1909;9:123-7.
14. Cacciatore B, Stenman UH, Ylostalo P. Diagnosis of ecto- pic pregnancy by vaginal ultrasonography in combination with a discriminatory serum hCG level of 1000 UI/I(IRP).
Br J Obstet.Gynecol 1990;97:904-8.
15. Altunyurt S, Guclu S, Kocaoglu S, Yorukoglu K. Primary ovarian pegnancy: a case report. T Klin J Gynecol Obst 2000;10:172-3.
